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Hindawi Publishing CorporationCase Reports in Emergency MedicineVolume 2011, Article ID 649548, 3 pagesdoi:10.1155/2011/649548

Case Report

Hypersensitivity Reaction to a Mosquito Bite in a Patient withChronic Myeloid Leukemia

Uri P. Dior,1 Shaden Salameh,1 Yonatan Gershinsky,2 and Ruth Stalnikowicz1

1 Department of Emergency Medicine, Hadassah University Hospital, Mount Scopus, P.O. Box 12000, 91120 Jerusalem, Israel2 Department of Internal Medicine, Hadassah University Hospital, Mount Scopus, P.O. Box 12000, 91120 Jerusalem, Israel

Correspondence should be addressed to Uri P. Dior, [email protected]

Received 1 July 2011; Accepted 9 August 2011

Academic Editors: H. David and W. Mauritz

Copyright © 2011 Uri P. Dior et al. This is an open access article distributed under the Creative Commons Attribution License,which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

A twenty-one-year-old male patient with an exaggerated hypersensitivity reaction to a mosquito bite presented to the departmentof emergency medicine for further evaluation. He was noted on physical examination to have splenomegaly. The hematologicalblood tests that were performed were compatible with chronic myeloid leukemia (CML). In this case, the mosquito bite heraldedthe diagnosis of CML.

1. Introduction

Exaggerated reaction to insect bites, mainly to mosquitoes,has been known to be associated with a few hematologicmalignancies. This phenomenon has been described mostlyin chronic lymphocytic leukemia [1–4] and mantle celllymphoma [5, 6], but has also been found rarely in acutelymphocytic leukemia, acute monocytic leukemia, and largecell lymphoma [7].

Usually, skin lesions appear months to years after thediagnosis of leukemia has been made and are unrelated tolaboratory findings, disease course, or therapy. To the bestof our knowledge, we describe the first reported case ofexaggerated reaction to insect bite associated with chronicmyelogenous leukemia. Furthermore, in this case the exag-gerated reaction led to the hematological diagnosis.

2. Case Report

A 21-year-old previously healthy male soldier presented tothe Department of Emergency Medicine (ED) for evaluationof swelling and pain in both legs. He stated that two eveningsearlier, he was stung in both feet by an insect, most likelya mosquito. Immediately after he was stung, he felt anitching sensation, without swelling or pain. The following

morning swelling appeared in both of his feet. The patientwas referred to a general practitioner who prescribed anantihistamine tablet (chlorpheniramine) and an unknowncream. There was no improvement and antibiotic treatmentwas prescribed the next day. Despite the antibiotics, theswelling continued to spread, resulting in his decision topresent to the ED. On presentation to the ED, he complainedof severe itching and pain and swelling spreading from hisfeet to his legs. He denied chest pain, shortness of breath,abdominal pain, back pain, or headache. He denied havingany fever for the previous two days.

His past medical history was unremarkable. A weekbefore his present admission to the ED, he had complainedof left upper quadrant pain. No prior history of allergy ordrug sensitivity was reported. The patient’s blood pressurewas 125/76 mmHg, pulse rate 92 beats/min, respiratory rate16 breaths/min, temperature 36.5◦C (97.1◦F), and oxygensaturation 97% on room air. His physical examination wasremarkable for pitting edema in his left foot and a mildswelling of his right foot. Diffuse erythemas, as well aslocalized erythematous papules consistent with insect bites,were observed on both legs. There was no induration orfluctuation suggestive of cellulitis or abscess. One smalland tender lymph node was palpated in his left groin. Noother enlarged lymph nodes were found. The abdominal

2 Case Reports in Emergency Medicine

examination demonstrated left upper quadrant tendernessand a palpable spleen. The remainder of the physical exam-ination was normal.

Hematologic studies revealed a white blood cell countof 44,300/mm3, a platelet count of 164,000/mm3, anda hemoglobin level of 14.1 gm/dL. The white blood celldifferential revealed 66.5% neutrophils, 25% lymphocytes,24.6% monocytes, and 2.6% basophiles. Electrolytes, bloodurea nitrogen level, glucose and renal function tests werenormal. Liver function tests were normal except for a mildelevation of the aspartate aminotransferase (AST) level upto 68 U/L (normal range 2–60 U/L). Lactate dehydrogenase(LDH) was markedly elevated up to 4,513 U/L (normalrange 300–620 U/L) and the uric acid was 487 umol/L (150–380 umol/L). Calcium, phosphorus, total protein, albumin,and creatinine kinase levels were normal.

Twelve hours after presenting to the ED and prior toinitiating any treatment, a new rash developed. The rash,involving the neck, upper chest, upper back, and the leftforearm was erythrodermic, well demarcated and non-pru-ritic. This rash resolved spontaneously 12 hours later.

A peripheral blood smear performed after the initialblood work showed elevated numbers of basophil, blasts, andimmature myeloid cells. The patient was transferred from theED to the hematology department for further evaluation.

An abdominal ultrasound was performed revealing sple-nomegaly measuring 17.5 cm with no evidence of infarction.Aspiration and biopsy of the bone marrow were performed.Direct visualization showed hyperplasia of all elements of thegranulocyte series. Maturation of the erythroid and granulo-cytic series was normal. An elevated number of megakary-ocytes and eosinophils were noticed and the myeloerythroidratio was high. Molecular diagnosis revealed 6.25E+5 copiesper reaction of BCR-ABL with a 94% BCR-ABL to ABLratio. Further genetic evaluation of the chromosomes derivedfrom the bone marrow sample showed a karyotype withthe reciprocal translocation T(9;22) (Q34;Q11) known asthe Philadelphia chromosome. The pathologic examinationof the biopsy sample demonstrated a significant increase ofthe myeloid lineage cells, composed mostly from immatureeosinophils. The diagnosis of intermediate Sokal scorechronic myelogenous leukemia was established.

3. Discussion

Chronic myelogenous leukemia (CML) is a hematologicalstem cell disorder characterized by excessive proliferation ofcells of the myeloid lineage. The genetic hallmark of CML isthe Philadelphia chromosome, which arises from a reciprocaltranslocation between chromosomes 9 and 22 that fusethe c-ABL tyrosine kinase gene on chromosome 9 and thebreakpoint cluster region (BCR) gene on chromosome 22.The disease is characterized by a chronic phase that can lastfor months or years and may exhibit few or no symptoms.Eventually, the chronic phase progresses to a more dangerousaccelerated or acute (blast crisis) phase, during which theleukemia cells acquire additional genetic changes, proliferatemore rapidly, and become resistant to apoptosis, and, if

untreated, ultimately cause the patient’s demise [8]. In arecent review [9], the median age at the time of diagnosiswas 60, and the youngest patient age was 28. Exposure toionizing radiation is the only known risk factor [10]. Theclinical findings at diagnosis of CML vary among reportedseries and also depend upon the stage of disease at diagnosis.Twenty to 50 percent of patients are asymptomatic, with thedisease first being suspected from routine blood tests. Amongsymptomatic patients, left upper quadrant abdominal painand early satiety, due to enlargement of the spleen with orwithout perisplenitis and/or splenic infarction, may occur[11]. Other frequent findings include splenomegaly (presentin 48% to 76% in two series), anemia (45% to 62%), whiteblood cell counts above 100,000/microL (52% to 72%), andplatelet counts above 600,000 to 700,000/microL (15% to34%) [10, 11].

A large local reaction to an insect bite is defined as edemaand erythema at the site of the bite, with a diameter greaterthan 10 cm and peaking in intensity at 24–48 hr. It can beevoked by a toxic mechanism, but it is generally believed thatan IgE-mediated or even cell-mediated pathogenesis occursin the majority of cases [12]. The prevalence of large localreactions to insect bites in the general population is variableacross studies, and it has been reported to range from 2.4 to10%, with a maximum of 26.4% [13].

The occurrence of unusual hypersensitivity phenomenahas been described in patients affected with a few hemato-logic disorders. The most frequent disorder where this phe-nomenon has been described is CLL [2, 3]. Weed [2] termedthese reactions as an “exaggerated delayed hypersensitivityto mosquito bites”. The hypothesis of an immunoallergicpathway, related to the onset of the cutaneous eruption, hasbeen proposed [14].

In our emergency department, blood tests are notroutinely drawn in patients with a presumed diagnosis ofallergic reaction. However, the finding of splenomegaly andleft upper quadrant tenderness raised the suspicion of anunderlying disease.

The diagnosis of CML is very rare at this young age, andhypersensitivity response to insect bite in CML has not beenpreviously reported. This combination seems unique, and wesuggest that when an exaggerated response to insect bite isencountered, further investigation for potential underlyingdisease is recommended.

References

[1] E. Robak and T. Robak, “Skin lesions in chronic lymphocyticleukemia,” Leukemia and Lymphoma, vol. 48, no. 5, pp. 855–865, 2007.

[2] R. I. Weed, “Exaggerated delayed hypersensitivity to mosquitobites in chronic lymphocytic leukemia,” Blood, vol. 26, pp.257–268, 1965.

[3] K. Asakura, M. Kizaki, and Y. Ikeda, “Exaggerated cutaneousresponse to mosquito bites in a patient with chronic lympho-cytic leukemia,” International Journal of Hematology, vol. 80,no. 1, pp. 59–61, 2004.

[4] M. D. Davis, C. Perniciaro, P. R. Dahl, H. W. Randle, M. T.McEvoy, and K. M. Leiferman, “Exaggerated arthropod-bite

Case Reports in Emergency Medicine 3

lesions in patients with chronic lymphocytic leukemia: a clin-ical, histopathologic, and immunopathologic study of eightpatients,” Journal of the American Academy of Dermatology, vol.39, no. 1, pp. 27–35, 1998.

[5] Z. Khamaysi, R. P. Dodiuk-Gad, S. Weltfriend et al., “Insectbite-like reaction associated with mantle cell lymphoma: clini-copathological, immunopathological, and molecular studies,”American Journal of Dermatopathology, vol. 27, no. 4, pp. 290–295, 2005.

[6] A. Kunitomi, Y. Konaka, and M. Yagita, “Hypersensitivity tomosquito bites as a potential sign of mantle cell lymphoma,”Internal Medicine, vol. 44, no. 10, pp. 1097–1099, 2005.

[7] A. Barzilai, D. Shpiro, I. Goldberg et al., “Insect bite-likereaction in patients with hematologic malignant neoplasms,”Archives of Dermatology, vol. 135, no. 12, pp. 1503–1507, 1999.

[8] T. Hunter, “Treatment for chronic myelogenous leukemia: thelong road to imatinib,” Journal of Clinical Investigation, vol.117, no. 8, pp. 2036–2043, 2007.

[9] D. Verma, H. M. Kantarjian, D. Jones et al., “Chronic myeloidleukemia (CML) with P190BCR−ABL: analysis of characteristics,outcomes, and prognostic significance,” Blood, vol. 114, no. 11,pp. 2232–2235, 2009.

[10] S. Faderl, M. Talpaz, Z. Estrov, S. O’Brien, R. Kurzrock, andH. M. Kantarjian, “The biology of chronic myeloid leukemia,”New England Journal of Medicine, vol. 341, no. 3, pp. 164–172,1999.

[11] D. G. Savage, R. M. Szydlo, and J. M. Goldman, “Clinicalfeatures at diagnosis in 430 patients with chronic myeloidleukaemia seen at a referral centre over a 16-year period,”British Journal of Haematology, vol. 96, no. 1, pp. 111–116,1997.

[12] J. E. Moffitt, D. B. Golden, R. E. Reisman et al., “Stinginginsect hypersensitivity: a practice parameter update,” Journalof Allergy and Clinical Immunology, vol. 114, no. 4, pp. 869–886, 2004.

[13] M. Severino, P. Bonadonna, and G. Passalacqua, “Largelocal reactions from stinging insects: from epidemiologyto management,” Current Opinion in Allergy and ClinicalImmunology, vol. 9, no. 4, pp. 334–337, 2009.

[14] C. Vassallo, F. Passamonti, R. Cananzi et al., “Exaggeratedinsect bite-like reaction in patients affected by oncohaema-tological diseases,” Acta Dermato-Venereologica, vol. 85, no. 1,pp. 76–77, 2005.

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